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本文引用的文献

1
Combined sequence and copy number analysis improves diagnosis of limb girdle and other myopathies.联合序列和拷贝数分析提高了肢带型和其他肌病的诊断能力。
Ann Clin Transl Neurol. 2023 Nov;10(11):2092-2104. doi: 10.1002/acn3.51896. Epub 2023 Sep 8.
2
Limb-Girdle Muscular Dystrophies Classification and Therapies.肢带型肌营养不良症:分类与治疗
J Clin Med. 2023 Jul 19;12(14):4769. doi: 10.3390/jcm12144769.
3
Causative variants linked with limb girdle muscular dystrophy in an Iranian population: 6 novel variants.在伊朗人群中与肢带型肌营养不良症相关的致病变异:6 个新的变异。
Mol Genet Genomic Med. 2023 Feb;11(2):e2101. doi: 10.1002/mgg3.2101. Epub 2022 Nov 14.
4
Mutational spectrum of autosomal recessive limb-girdle muscular dystrophies in a cohort of 112 Iranian patients and reporting of a possible founder effect.常染色体隐性遗传肢带型肌营养不良症 112 例伊朗患者的突变谱及可能的 founder 效应报道。
Orphanet J Rare Dis. 2020 Jan 14;15(1):14. doi: 10.1186/s13023-020-1296-x.
5
Genetic variability in Iranian limb-girdle muscular dystrophy type 2B patients: An evidence of a founder effect.伊朗肢带型肌营养不良症 2B 患者的遗传变异性:一个奠基者效应的证据。
Mol Genet Genomic Med. 2019 Dec;7(12):e1029. doi: 10.1002/mgg3.1029. Epub 2019 Nov 6.
6
Genetic landscape and novel disease mechanisms from a large LGMD cohort of 4656 patients.来自4656例患者的大型肢带型肌营养不良症队列的遗传图谱和新的疾病机制。
Ann Clin Transl Neurol. 2018 Dec 1;5(12):1574-1587. doi: 10.1002/acn3.649. eCollection 2018 Dec.
7
Prevalence, pathological mechanisms, and genetic basis of limb-girdle muscular dystrophies: A review.肢带型肌营养不良症的患病率、病理机制和遗传基础:综述。
J Cell Physiol. 2019 Jun;234(6):7874-7884. doi: 10.1002/jcp.27907. Epub 2018 Dec 7.
8
Untangling the complexity of limb-girdle muscular dystrophies.理清肢带型肌营养不良症的复杂性。
Muscle Nerve. 2018 Aug;58(2):167-177. doi: 10.1002/mus.26077. Epub 2018 Feb 7.
9
Limb Girdle Muscular Dystrophy Type 2E Due to a Novel Large Deletion in SGCB Gene.由于SGCB基因出现新型大片段缺失导致的2E型肢带型肌营养不良症
Iran J Child Neurol. 2017 Summer;11(3):57-60.
10
Mutational spectrum of Chinese LGMD patients by targeted next-generation sequencing.通过靶向二代测序技术分析中国肢带型肌营养不良症患者的突变谱。
PLoS One. 2017 Apr 12;12(4):e0175343. doi: 10.1371/journal.pone.0175343. eCollection 2017.

伊朗肢带型肌营养不良患者突变谱的调查。

A survey on mutation spectrum in Iranian patients with limb-girdle muscular dystrophies.

作者信息

Khalilian Sheyda, Fathi Mohadeseh, Tangestani Raheleh, Larki Pegah, Sayad Arezou, Ghafouri-Fard Soudeh, Miryounesi Mohammad

机构信息

Student Research Committee, School of Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Iran.

Department of Medical Genetics, Shahid Beheshti University of Medical Sciences, Tehran, Iran.

出版信息

Hum Genomics. 2025 May 13;19(1):54. doi: 10.1186/s40246-025-00771-4.

DOI:10.1186/s40246-025-00771-4
PMID:40361203
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12076818/
Abstract

Limb-girdle muscular dystrophies (LGMD) designate diverse types of muscular dystrophies that predominantly affect proximal skeletal muscles. Although both autosomal recessive and dominant forms exist, the majority of cases are inherited in an autosomal recessive manner. Since the spectrum of genetic variants that cause this disorder is quite broad, next-generation sequencing techniques are the best diagnostic tools for LGMD. In this study, we provide an overview of mutation spectrum of LGMD-related genes in the Iranian patients using whole exome sequencing. Notably, CAPN3 and LAMA2 genes were the genes encompassing the highest frequencies of pathogenic or likely pathogenic variants in this cohort. Pathogenic and likely pathogenic variants were identified in CAPN3 gene in total of 10 cases out of 48 cases tested (20%). In addition, different variants in each of POMGNT1 and TTN genes were detected in five and four patients, respectively. Three patients had DYSF variants (6%). While the inheritance of the majority of cases was supposed to be in an autosomal recessive manner, in three cases, the disease inheritance was best explained by the dominant type (c.947 C > T variant in the DNAJB6, c.746G > A variant in the LMNA, and c.1417G > A variant in the TNPO3). The current study broadens the spectrum of LGMD-related mutations among Iranian patients and facilitates genetic counseling in the affected families.

摘要

肢带型肌营养不良症(LGMD)指的是多种主要影响近端骨骼肌的肌营养不良症。虽然存在常染色体隐性和显性两种形式,但大多数病例是以常染色体隐性方式遗传的。由于导致这种疾病的基因变异谱相当广泛,下一代测序技术是诊断LGMD的最佳工具。在本研究中,我们使用全外显子组测序概述了伊朗患者中LGMD相关基因的突变谱。值得注意的是,在该队列中,CAPN3和LAMA2基因是包含致病性或可能致病性变异频率最高的基因。在48例检测病例中,共有10例(20%)在CAPN3基因中鉴定出致病性和可能致病性变异。此外,分别在5例和4例患者中检测到POMGNT1和TTN基因中的不同变异。3例患者有DYSF变异(6%)。虽然大多数病例的遗传方式被认为是常染色体隐性,但在3例病例中,疾病遗传最好用显性类型来解释(DNAJB6中的c.947 C>T变异、LMNA中的c.746G>A变异和TNPO3中的c.1417G>A变异)。本研究拓宽了伊朗患者中LGMD相关突变的谱,并有助于为受影响家庭提供遗传咨询。